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Major respiratory catastrophe associated with iatrogenic airway injury during the Sistrunk operation is a rare event. A three-year-old patient underwent thyroglossal duct cyst removal under general anaesthesia. An iatrogenic thyroid cartilage injury occurred in the intraoperative period, and it was repaired primarily. Later, in the post-anaesthesia care unit, the patient developed subcutaneous emphysema in the neck and face, and then pneumomediastinum and bilateral pneumothoraces. The patient was managed with bilateral chest drains and endotracheal intubation, and he required mechanical ventilation for three days. So, even after repair of a recognised iatrogenic airway injury associated with the Sistrunk operation, it may be necessary to continue positive pressure ventilation in the postoperative period to avoid serious respiratory complications. [ABSTRACT FROM AUTHOR]